Posterior Instrumentation without Curettage Promotes Rapid Restoration of Adult Spinal Langerhans Cell Histiocytosis

Abstract

Introduction: Adult spinal Langerhans cell histiocytosis (LCH) presents a treatment challenge due to ongoing controversies. Traditional approaches such as curettage with bone grafting and internal fixation are preferred for severe cases involving mechanical instability, neurological deficits, or deformity. This study aimed to explore the efficacy of a customized approach involving simple posterior instrumentation without curettage or bone grafting in treating adult spinal LCH.

Methods: This retrospective study analyzed a prospectively maintained database of all spine surgeries conducted at our institute from April 2013 to December 2020. Adult patients (age≥20) diagnosed with LCH were included. We assessed surgical methods, adjuvant therapy, and clinical results, such as perioperative progression of disease, symptoms, and recurrence.

Results: Four male patients aged between 21 and 28, each with a single spinal LCH lesion (T6, T5, and C5) except one case (T5 and T7), were treated. Diagnoses were confirmed via biopsy (two open, two needle biopsies). Whole-body computed tomography or bone scintigraphy revealed no additional LCH lesions in any patient, except in one patient with a small lung nodule. All patients presented with severe back or neck pain and pathological fractures at the affected vertebra. Thoracic LCH cases received percutaneous pedicle screw fixation, while the cervical case was managed with conventional posterior instrumentation using lateral mass screws. After surgery, all patients experienced significant pain relief, halted bone lysis, and rapid new bone formation. One patient underwent chemotherapy postsurgery. Over 3 years of follow-up, imaging studies revealed no recurrences of the disease.

Conclusions: Posterior instrumentation, without the need for curettage or bone grafting, is a promising surgical treatment for adult spinal LCH. This method may effectively halt lesion progression, prevent spinal deformity, and avert neurological deficits in the patients with progressive spine lesion where conservative treatment may not adequately prevent vertebral fractures.

Keywords: Adult spinal LCH; Curettage; Langerhans cell histiocytosis (LCH); Percutaneous pedicle screw; Posterior instrumentation; Spine tumor.

Figure 1.

Imaging study of Case 1: A, T2-weighted (left) and T1-weighted (right) magnetic resonance imaging (MRI) displaying an abnormal lesion in the T6 vertebral body, lamina, and epidural space. B, T2-weighted axial MRI highlighting the abnormal lesion extending into the epidural and perivertebral spaces (indicated by white arrowheads). C, Lateral X-ray postfixation surgery, showing the surgical outcome. D and E, T2-weighted MRI scans taken 7 days postsurgery, revealing a significant reduction in the size of the epidural lesion.

Figure 2.

This series shows the progression and treatment response of the bone lysis in Case 1. Following the posterior instrumentation with the percutaneous pedicle screw system, there was a cessation of the rapid bone lysis. New bone formation was observed as early as 6 weeks postsurgery. The final CT scan, taken 3 years postoperatively, reveals a vertebra that appears almost normal.

Figure 3.

Imaging study of Case 2. A, Fat suppression T2-weighted (left) and T1-weighted (right) magnetic resonance imaging (MRI) showing an abnormal lesion in the T5 vertebra of Case 2. B, Fat suppression T2-weighted axial MRI revealing perivertebral and epidural lesions. C, A small nodule observed in the right lung. D, X-ray taken after the fixation surgery, showing the postoperative status. E, Postoperative X-ray taken 2 years later, after the removal of the instrumentation.

Figure 4.

This series shows the progression and subsequent response to treatment of bone lysis in Case 2. Following the posterior instrumentation using the percutaneous pedicle screw system, a halt in the rapid progression of bone lysis was observed. Seven months after operation, significant bone reconstitution is evident, although some deformation of the endplate persists, as was present prior to the operation.